Clinical management of patients with cystic echinococcosis has been established at our hospital through interdisciplinary cooperation with the Department of Surgery, the Department of Diagnostic and Interventional Radiology (with weekly radiological conferences), the Department of Parasitology and the Thoraxklinik.

Today we are a national clinical reference centre for patients with cystic echinococcosis; in international terms, we are a well-established partner in the field of clinical echinococcosis research.

- R. Gilman, Johns Hopkins Bloomberg School of Public Health, Baltimore MD, US

- M. Zwahlen, University of Bern, Switzerland

The specific aims of the R34 planning grant are to develop the protocol, SOPs and related trial documents for a Phase 2 clinical trial of stage-specific treatment of cystic echinococcosis (EchinoTrial) and to select the trial sites in Peru. The main study is a pragmatic randomised controlled trial to determine the efficacy, safety and relapse rate of the four currently available treatment modalities of uncomplicated hepatic CE cysts stratified by cyst stage as defined by the WHO ultrasound classification 2002 [Phase 2 stage-specific clinical trial (main study)]. Given the extraordinary clinical complexity of CE, treatment standardization requires the exclusion of patients with complicated cysts in need of individualized therapy. Patients with complicated CE cysts (e.g. cysto-biliary fistulas, cyst rupture, secondary bacterial infection) will be investigated in a prospective observational non-comparative study arm (substudy). The trial site in Peru offers access to a highly endemic population for CE. The expected outcome of this trial will create a solid evidence base for the allocation of available treatment modalities on cyst stage level, will help to avoid unnecessary treatment of inactive cysts and will define the long term prognosis after treatment (relapse rate).

The submission of the trial protocol and application for a "Clinical trial implementation grant" was due in January 2012.

Drug therapy with benzimidazoles is one of four options available for the treatment of cystic echinococcosis, which has been investigated in a great number of clinical studies. However, individual studies are not large enough to provide reliable estimates regarding the effect of treatment. Heterogeneity of the inclusion criteria and the study endpoints does not allow for meta-analysis. Moreover, most of the studies published to date do not allow for the stratification of the treatment results according to cyst staging determined by ultrasound. However, this is a classification that seems indispensable today regarding the choice of treatment modality and the monitoring of treatment success.

Individual patient data is reanalysed to (1) gather evidence regarding the efficacy of benzimidazole treatment based on the data available and (2) plan a multicentre clinical study to compare the four treatment modalities currently available stratified by cyst stage, with benzimidazoles being the only drug therapy.

From country level to a pan-European perspective: a coordinated approach to controlling cystic echinococcosis (EchinoNET)

The project aims to develop a standardised methodology for transnational data collection and data presentation in the fields of surveillance, prevention and clinical management of cystic echinococcosis (CE); to collect and assess data on prevention measures in the endemic partner countries as well as collect and assess data on the four treatment modalities surgery, PAIR, drug treatment and ‘watch & wait’ from endemic (Mediterranean partner countries) and non-endemic (EU) countries; to make recommendations on surveillance, prevention, treatment and quality management.

Data collection regarding the diagnosis, treatment and control of cystic echinococcosis in the EchinoNET partner countries as well as data analysis have been completed. EchinoNET is currently expanding its network to include CE-endemic countries in Latin America with the aim to compare the four treatment modalities currently available in a multicentre clinical study stratified by cyst stage. This would be the first clinical study on this neglected disease to be large enough and carried out to the required standards.